Case Report: Urethral Diverticulum after Tubularised Incised Plate Repair of Distal Hypospadias: An Uncommon Complication.

Introduction A urethral diverticulum can be defined as a pocket that forms from the lining of the urethra and protrudes into the surrounding tissue, a condition which causes voiding dysfunction and may result as a rare complication of hypospadias repair surgery. Case report We report the case of a 2-year-old child who presented to us in 2019 complaining of a thin forceful stream, ballooning of the ventral aspect of the penis while voiding, and post-void dribbling. He has a history of undergoing a tubularised incised plate urethroplasty for distal penile hypospadias at 18-months-old. Ultrasound showed increased post-void residual volume and cystourethroscopy confirmed a urethral diverticulum extending from the subcorona to the base of the penis. The patient underwent partial excision of diverticulum, urethroplasty, and meatoplasty. He was followed-up 3 months later with complete resolution of his symptoms and a normal urinary stream with no urethral ballooning or dribbling. Conclusion Urethral diverticulum may present as a complication post hypospadias repair. Although it is rare, we believe that it is important for the patient’s parents to understand the possibility and know of the signs and symptoms in addition to attending regular outpatient clinic appointments in order to facilitate early management if needed. Furthermore, it is highly important for physicians to assess newborns for hypospadias before carrying out circumcision as it is a contraindication for the procedure.


Introduction
A urethral diverticulum can be defined as a pocket that forms from the lining of the urethra and protrudes into the surrounding tissue, a condition which causes voiding dysfunction and may result as a rare complication of hypospadias repair surgery.

Case report
We report the case of a 2-year-old child who presented to us in 2019 complaining of a thin forceful stream, ballooning of the ventral aspect of the penis while voiding, and post-void dribbling.He has a history of undergoing a tubularised incised plate urethroplasty for distal penile hypospadias at 18-months-old.Ultrasound showed increased postvoid residual volume and cystourethroscopy confirmed a urethral diverticulum extending from the subcorona to the base of the penis.The patient underwent partial excision of diverticulum, urethroplasty, and meatoplasty.He was followed-up 3 months later with complete resolution of his symptoms and a normal urinary stream with no urethral ballooning or dribbling.Any reports and responses or comments on the article can be found at the end of the article.

Introduction
Urethral diverticulum -an out-pouching of the urethra into surrounding tissues -is an important but uncommon complication of hypospadias repair. 1 Studies suggest that it comprises 0.3% of postoperative complications and is mainly associated with proximal hypospadias. 2,3Factors predisposing to its formation include proximal defect, oversized neourethra, poorly supporting tissue covering urethroplasty, and distal urethral stricture. 2,3We present a case of postoperative urethral diverticulum formation after distal hypospadias repair and discuss possible treatment options.

Case report
The child was one-month-old (Figure 1) when referred to our centre in 2019 for review after undergoing Plastibell circumcision.On examination, he was circumcised with a hypospadiac meatus located at distal penile position (Figure 2).
At 18 months of age, he underwent a tubularised incised plate urethroplasty at an external paediatric urology centre with an excellent outcome (Figure 3).
One year later, he presented with typical signs and symptoms of a urethral diverticulum and meatal stenosis; thin forceful stream, ballooning of the ventral aspect of the penis during voiding, significant post-void dribbling, and need for manual emptying of the diverticulum (Figure 4).
Ultrasound showed diffuse bladder wall thickening with significant post-void residual volume.Ascending urethrogram confirmed the diagnosis of meatal stenosis with proximal diverticulum (Figure 5).Patient underwent cystourethroscopy which confirmed a diverticulum extending from the subcorona to the base of the penis.The diverticulum was opened through a ventral midline incision.A midline strip of 16 mm width of mucosa was preserved, and lateral mucosa was excised preserving the underlying dartos tissue (Figure 6).
The urethroplasty was closed in two layers and was supported by double-breasted Dartos fascia.At the end of the procedure, standard meatoplasty was performed for meatal stenosis.Three months following repair, the child had a normal thick continuous stream with no post-void dribbling or ballooning of the urethra.REVISED Amendments from Version 1 -The title of the article has been changed to more accurately describe the case reported.
-A few spelling mistakes were corrected.

Discussion
Urethral diverticulum is an uncommon complication of hypospadias surgery which has several risk factors that could influence its incidence such as a large neourethra, inadequate supporting tissue post-urethroplasty, and a distal urethral stricture. 2,3It can be diagnosed via clinical evaluation and confirmed with cystourethrography and cystoscopy.Partial excision of diverticulum and urethroplasty is an acceptable form of treatment for such cases, as the final aim is to restore a urethra with normal diameter and pressure that is also well supported with enough tissue and no distal obstruction. 2,4In the presented case, the diagnosis of hypospadias was missed at birth and a circumcision was carried out though circumcision is contraindicated in these cases.This certainly requires more awareness since the repair of hypospadias in uncircumcised children is technically easier and has a better outcome.Additionally in this case, excessive circumcision resulted in a very wide meatus along with a severe deficiency in the ventral skin and the underlying supportive tissue which possibly complicated the repair and played an important role in the formation of the diverticulum.Meatal stenosis is another factor that predisposed to diverticulum formation in this child, and it was most likely a postsurgical complication as it can occur after any hypospadias surgery.Therefore, regular follow-up after hypospadias repair is of vital importance for early diagnosis and management of meatal stenosis.

Conclusion
Meatal stenosis and urethral diverticulum are amongst the possible complications of hypospadias repair.The repair of urethral diverticula is challenging, and the outcome depends on the creation of a well-supported and normal-sized urethra with no distal obstruction.Furthermore, parents should be educated about the signs and symptoms of meatal stenosis and the need for strict and regular follow-up to diagnose and manage any complication as early as possible.It is important to carefully examine the child for hypospadias before circumcision as it is contraindicated.

Consent
We discussed and explained to the patient's father about the writing and publication of this case report.He had the opportunity to ask questions about everything related to this process, and understood that no identifying information related to the patient will be shared; Written informed consent for the publication of patient details and images was obtained from the patient's father.

Johannes Wirmer
Sana Klinikum, Offenbach, Germany The Report describes a case of urethral diverticulum after tubularized-inzised plate repair (TIP) of a distal Hypospadias.The Hypospadias repair was performed at the age of 18 month, but had a circumcision as a newborn.From the picture taken before the hypospadias repair, he most likely had a MIP-Hypospadias (megameatus intact prepuce).
The here described developement of an urethral diverticulum after repair of distal hypospadias is an important complication of the TIP-technique.Even though it has been described before ( Lee H, et al., 2006) [Ref 1], the common assumption is still that it mainly occurs after flap urethroplasty.This article is an important contribution to drawing attention to the complications of tip correction.
Small suggestions for corrections would be: Title: Case Report: Urethral Diverticulum after TIP-Repair of distal hypospadias: a not so uncommon complication In addition, tabularized incised plate urethroplasty is mentioned several times throughout the text, where it should correctly be called TUBULARIZED plate urethroplasty.Is the case presented with sufficient detail to be useful for other practitioners?Yes

Figure 1 .
Figure 1.A timeline of the patient's presentations and the procedures he underwent.

Figure 3 .
Figure 3. Ventral view of the penis after tubularised incised plate urethroplasty.

Figure 2 .
Figure 2. Ventral view of the penis (excessively circumcised with a distal penile patulous meatus).

2 .
Horton CE, Horton CE: Complications of hypospadias surgery.Clin Plast Surg.1988; 15 (3): 371-9 PubMed Abstract 3. Snyder CL, Evangelidis A, Snyder RP, Ostlie DJ, et al.: Management of urethral diverticulum complicating hypospadias repair.J Pediatr Urol.2005; 1 (2): 81-3 PubMed Abstract | Publisher Full Text Is the background of the case's history and progression described in sufficient detail?Yes Are enough details provided of any physical examination and diagnostic tests, treatment given and outcomes?Yes Is sufficient discussion included of the importance of the findings and their relevance to future understanding of disease processes, diagnosis or treatment?Partly Is the case presented with sufficient detail to be useful for other practitioners?Yes Competing Interests: No competing interests were disclosed.Reviewer Expertise: Pediatric surgery I confirm that I have read this submission and believe that I have an appropriate level of expertise to confirm that it is of an acceptable scientific standard, however I have significant reservations, as outlined above.Reviewer Report 05 April 2024 https://doi.org/10.5256/f1000research.162472.r261654© 2024 Wirmer J.This is an open access peer review report distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

References 1 .
Lee H, Han S: Surgical Correction of Post-hypospadias Repair Urethral Diverticulum and Stricture using Rotational Flap.Korean Journal of Urology.2006; 47 (11).Publisher Full TextIs the background of the case's history and progression described in sufficient detail?YesAre enough details provided of any physical examination and diagnostic tests, treatment given and outcomes?YesIs sufficient discussion included of the importance of the findings and their relevance to future understanding of disease processes, diagnosis or treatment?Partly